Suppr超能文献

伴有免疫缺陷的部分白化病:格里谢利综合征:1例报告并文献复习

Partial albinism with immunodeficiency: Griscelli syndrome: report of a case and review of the literature.

作者信息

Mancini A J, Chan L S, Paller A S

机构信息

Department of Pediatrics, Northwestern University Medical School, Chicago, Illinois 60614, USA.

出版信息

J Am Acad Dermatol. 1998 Feb;38(2 Pt 2):295-300. doi: 10.1016/s0190-9622(98)70568-7.

Abstract

Partial albinism with immunodeficiency (Griscelli syndrome) is an uncommon disorder characterized by pigmentary dilution and variable immunodeficiency. Features include a silvery-gray sheen to the hair, large clumped melanosomes in hair shafts, and prominent mature melanosomes in cutaneous melanocytes with sparse pigmentation of adjacent keratinocytes. Immunologic abnormalities most often include impaired natural killer cell activity, absent delayed-type hypersensitivity, and impaired responses to mitogens. Impaired helper T cell function and hypogammaglobulinemia have also been described. The syndrome can be differentiated from Chediak-Higashi syndrome by pathognomonic light and electron microscopic features in skin and hair, and absence of consistent granulocyte abnormalities, but similarly carries a poor prognosis without bone marrow transplantation. We describe a patient with Griscelli syndrome who presented with hepatosplenomegaly, hepatitis, pancytopenia, and silvery hair in the newborn period.

摘要

伴有免疫缺陷的部分白化病(格里塞利综合征)是一种罕见的疾病,其特征为色素稀释和可变的免疫缺陷。特征包括头发有银灰色光泽、毛干中有大的聚集黑素小体、皮肤黑素细胞中有突出的成熟黑素小体且相邻角质形成细胞色素沉着稀疏。免疫异常最常包括自然杀伤细胞活性受损、迟发型超敏反应缺失以及对有丝分裂原的反应受损。也有辅助性T细胞功能受损和低丙种球蛋白血症的描述。该综合征可通过皮肤和头发的特征性光镜和电镜表现与切迪阿克 - 东综合征相鉴别,且无一致的粒细胞异常,但同样在无骨髓移植的情况下预后较差。我们描述了一名患有格里塞利综合征的患者,该患者在新生儿期出现肝脾肿大、肝炎、全血细胞减少和银发。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验